Introduction: Pediatric dental treatment utilizing an air-driven handpiece and triplex syringe in the operating room has been common practice for many decades. Adverse effects with compressed air are rarely reported in the dental community but can cause serious complications for pediatric patients in the unlikely event they occur.

Case Report: We present a case of subcutaneous emphysema of a 4-year-old presenting for dental rehabilitation under general anesthesia, which was further complicated by the presence of a ventriculoperitoneal (VP) shunt and the patient's neuroatypical status. The subcutaneous emphysema was apparent at case completion with right unilateral swelling in the cervicofacial area extending from the infratemporal space to the periorbital buccal region. The most probable etiology was compressed air from a high-speed handpiece dissecting through infected tissue during a crown preparation. Subcutaneous emphysema was managed by diagnostic radiographs, antibiotics coverage, and hospital observation due to patient's multiple health factors.

Conclusion: Subcutaneous emphysema appears to be a rare event in pediatric dental care; however, there is increased risk for complications in the unintentional occurrence. Subcutaneous emphysema is an important differential for facial swellings. Medically complex patients, such as this case, should involve a multidisciplinary team approach due to the proximity of the VP shunt.

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